Objective: Propriospinal myoclonus (PSM) is a rare disorder with repetitive, usually flexor arrhythmic brief jerks of the trunk, hips, and knees in a fixed pattern. It has a presumed generation in the spinal cord and diagnosis depends on characteristic features at polymyography. Recently, a historical paradigm shift took place as PSM has been reported to be a functional (or psychogenic) movement disorder (FMD) in most patients. This review aims to characterize the clinical features, etiology, electrophysiologic features, and treatment outcomes of PSM.Methods: Re-evaluation of all published PSM cases and systematic scoring of clinical and electrophysiologic characteristics in all published cases since 1991.Results: Of the 179 identified patients with PSM (55% male), the mean age at onset was 43 years (range 6-88 years). FMD was diagnosed in 104 (58%) cases. In 12 cases (26% of reported secondary cases, 7% of total cases), a structural spinal cord lesion was found. Clonazepam and botulinum toxin may be effective in reducing jerks.Conclusions: FMD is more frequent than previously assumed. Structural lesions reported to underlie PSM are scarce. Based on our clinical experience and the reviewed literature, we recommend polymyography to assess recruitment variability combined with a Bereitschaftspotential recording in all cases. Propriospinal myoclonus (PSM) is a rare hyperkinetic movement disorder involving the axial muscles.1 Brown et al. 1 were the first to describe the clinical phenomenology based on 3 cases. PSM is characterized by painless, usually flexor arrhythmic jerks of the trunk, hips, and knees, which are often stimulus sensitive and typically increase when supine.1-3 Based on the initial publications, PSM had a presumed generation in the spinal cord and diagnosis depended on characteristic features at polymyography. [1][2][3][4] The myoclonic activity in PSM was assumed to originate from a spinal generator and spread up and down the spinal cord via intrinsic propriospinal pathways, and these axial jerks were therefore termed propriospinal myoclonus.1 Electrophysiologic features of PSM were published after the first clinical descriptions and included a fixed pattern of muscle activation, a slow conduction velocity (5-15 m/s) consistent with spread within the propriospinal tract, EMG burst duration of less than 1,000 ms, synchronous activation of agonist and antagonist muscles, and no facial involvement. 4 Until recently, patients were subdivided into idiopathic PSM (i.e., primary, no underlying cause found) and secondary PSM. However, the diverse etiology of this disorder is currently highlighted by several movement disorders specialists, including our centres.5-7 A functional etiology was reported to underlie several PSM cases. Psychogenic or functional movement disorders (FMD) are common and characterized by abnormal movements inconsistent with a known organic etiology. 8